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Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015 Copyright 2015 American Speech-Language-Hearing Association
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Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015
Aim
The primary aim of this study was to use objective
statistical methods to identify one or more quantitative
measures of speech that reproduce the expert classification
of children as having CAS or not. This approach has potential to provide a clinically feasible assessment protocol that
reliably identifies features of CAS and reduces reliance on
expert opinion.
Method
This study formed part of a larger clinical trial:
Unique Trial Number: U1111-1132-5952, Trial Registration
Number: ACTRN12612000744853. The research protocol
was approved by the Human Research Ethics Committee of
the University of Sydney (12924) and is published (Murray,
McCabe, & Ballard, 2012).
This diagnostic study was conducted in three phases
to address the research aim. First, children were recruited
and assessed according to the protocol below. Second, the
first two authors (E. M. and P. M.) judged presence or absence of CAS by listening to speech samples from the assessment protocol and completing two published checklists
of CAS characteristics. Third, the first author (E. M.) and
independent raters blinded to the diagnosis of CAS generated 24 quantitative measures from assessment data. These
Participants
Participants were recruited via a website advertisement
and flyers as well as e-mails and listserv posts to speechlanguage pathologists (SLPs), inviting them to volunteer for
a research treatment study. The treatment component of this
study is not discussed in this article. The inclusion criteria for
participants were (a) a clinical diagnosis of suspected CAS
by a community-based SLP, (b) age between 4 and 12 years,
(c) no previously identified language comprehension difficulty, (d) normal or adjusted-to-normal hearing and vision,
(e) native English speaker, and (f ) no other developmental
diagnoses not associated with CAS (e.g., intellectual disability,
autism, cerebral palsy). All participants had received speechlanguage pathology intervention prior to this study, and
all had normal hearing. Participants needed to attend the
on-campus clinic at the University of Sydney, Australia, to
participate. Parents or caregivers provided informed consent
on behalf of the children prior to participation.
Of the 72 participants who inquired regarding the
study, 47 passed the initial screening. All 47 were AustralianEnglish speaking, 33 (70%) were boys, 14 were girls (30%),
and the average age was 70.5 months (SD = 25.7 months).
No information on race, ethnicity, or socioeconomic status
was collected.
Clinical Assessment
The first author responded to all inquiries to participate in the study and followed a two-tiered assessment
protocol (see Figure 1). The first tier involved screening of
parents or caregivers and SLPs by phone and previous
speech pathology reports to determine the childs suitability
for entry into the study. The criteria were applied conservatively, in that children with another developmental disorder that could be associated with symptoms or consequences
of CAS (e.g., selective mutism) were included. For those
who passed the initial screening, the second tier involved administration of a 2-hour speech and language assessment
battery. Responses to these tests were used for all analyses
of CAS features and to determine the presence of comorbid
conditions such as dysarthria or language impairment. The
assessment battery was video- and audio-recorded using
a Sony IC Recorder ICD-UX71F and either an Echo Layla
24/96 multitrack recording system, Marantz PMD660 solidstate recorder, or Roland Quad-Capture UA-55 at the sampling rate 48000 Hz with 16-bit resolution with an AKG
C520 headset microphone at 5 cm mouth-to-microphone
distance.
45
Figure 1. Results of differential diagnostic process. Participants with childhood apraxia of speech (CAS) shown in white, comorbid CAS in yellow,
and non-CAS or excluded in blue.
the diagnostic assessment utilized five published tests commonly available in clinical settings and culturally appropriate
for Australian children (see Table 1). The five tests were the
following:
1.
2.
3.
4.
5.
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Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015
Table 1. Assessment tasks and diagnostic features used for assigning an expert diagnosis of childhood apraxia of speech.
ASHA consensus-based
feature list a
1. Inconsistent errors on
consonants and vowels in
repeated productions of
syllables or words
2. Lengthened and disrupted
coarticulatory transitions
between sounds and syllables
3. Inappropriate prosody,
especially in the realization of
lexical or phrasal stress
Not in list
Quantitative Measures
Quantitative measures were extracted from the five
tests administered in the clinical assessment. Table 2 lists
the 24 measures generated and the associated assessment
task and CAS checklist feature. For measures that were
not standard scores from norm-referenced tools, nine raters
47
48
Table 2. Extracted measures from assessment tasks completed with means and standard deviations for the CAS, CAS+, and non-CAS groups.
Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015
CAS (n = 28)
Assessment task
Case history
DEAP Inconsistency
subtesta
Single-Word Test of
Polysyllablesb
Voicing errors
None
None
None
Increased difficulty with longer or more
phonetically complex wordsg
Slow rateg
Lengthened and disrupted coarticulatory
transitions between sounds and
syllablesf and difficulty achieving initial
articulatory configurations and
transitions into vowelsg
Groping (nonspeech)g
Slow DDK rateg
Slow DDK rateg
Slow DDK rateg
Slow DDK rateg
Slow DDK rateg
None
CELFe
None
None
None
None
None
Measure
Age in months
Percentage inconsistency (across three
repetitions of 25 words)a
Percentage of lexical stress matches
(relative to gloss)h
Distortion occurrences (out of a possible
328 phones)h
Syllable segregation occurrences (out of a
possible 114 opportunities)i
Intrusive schwa occurrences (out of a
possible 15 clusters)i
Voicing error occurrences (out of a possible
90 opportunities)h
PPCh
PCC-Rh,j
PVC on polysyllable testh
Magnitude of change score: NPC on 12
monosyllables/NPC on 12 polysyllables
(>1 = difficulty with polysyllables)
Articulation rate (syllables per minute)k
Presence of false articulatory starts and
restarts and/or inaudible within-speech
groping and/or audible within-speech
groping and/or hesitations (min. = 0,
max. = 1)l,m
Presence of nonspeech groping in lip and
tongue oral function tasks (min. = 0,
max. = 1)
/p/ rate over 3 s on two trialsd
/t/ rate over 3 s on two trialsd
/k/ rate over 3 s on two trialsd
/ptk/ rate over 3 s on two trialsd
/ptikek/ rate over 3 s on two trialsd
Accuracy on /ptk/ DDK task on
two trialsd
Oral structure scored
Oral function scored
Maximum Phonation Timed
Receptive Language Scoree
Expressive Language Scoree
CAS+ (n = 4)
Non-CAS (n = 15)
SD
SD
SD
66.5
63.8
24.8
12.8
72.5
75.0
26.0
15.1
77.3
39.9
27.6
24.5
9.8
9.1
16.3
17.5
67.3
22.4
39.4
5.8
45.2
8.8
46.24
22.9
30.2
8.8
8.5
8.4
1.2
2.1
1.1
1.3
0.5
1.0
0.0
0.0
3.4
4.0
6.0
5.0
1.7
1.5
52.2
54.0
50.5
1.23
15.0
20.0
11.4
0.2
24.3
22.8
24.8
2.52
20.6
23.8
19.0
1.9
73.1
70.9
75.1
2.07
26.3
26.4
27.3
2.91
1.7
0.9
1.0
0.4
0.9
1.0
0.6
0.0
2.6
0.1
1.0
0.3
0.5
0.5
0.5
0.6
0.0
0.0
3.6
3.4
2.9
0.9
1.0
48.5
1.0
1.0
1.2
0.6
0.4
22.6
3.0
2.3
1.8
0.5
0.7
23.8
1.7
1.3
1.0
0.1
0.4
9.0
4.5
4.4
4.3
1.4
1.3
75.9
2.0
1.7
1.9
0.8
0.7
23.2
23.4
76.6
9.6
100.3
85.5
.91
13.8
5.5
12.2
20.1
21.0
60.0
6.8
65.5
55.5
2.6
12.4
4.1
18.4
7.3
21.8
88.5
12.9
91.4
79.7
2.4
18.7
6.7
20.5
19.2
Note. CAS = childhood apraxia of speech; M = mean; SD = standard deviation; PPC = percentage phonemes correct; PCC-R = percentage consonants correctrevised; PVC = percentage
vowels correct; NPC = number of phonemes correct; CELF = Clinical Evaluation of Language Fundamentals (Semel, Wiig, & Secord, 2006; Wiig, Secord, & Semel, 2006).
a
Dodd et al. (2002). bGozzard et al. (2004, 2008). cMcLeod (1997). dRobbins and Klee (1987) includes some norms. eSemel et al. (2006) with norms. fASHA (2007b). gShriberg, Potter, et al.
(2009). hComputerized Profiling (Long et al., 2006). iCounted by hand from transcription. jShriberg, Austin, Lewis, McSweeny, and Wilson (1997). kLogan et al. (2011) includes norms.
l
McNeil et al. (2009). mDuffy (2012).
were employed to make measurements blinded to the childrens expert diagnosis of CAS presence or absence for reliability purposes. The raters were qualified SLPs with an
average of 3 years of clinical experience. The first author
trained each rater on the methods of measurement until a
minimum of 85% interrater reliability was achieved on a
training sample not included in the study. Raters were randomly assigned samples from 12 to 13 children (M = 12.70,
SD = 4.83). Each participants results were rated by at least
two raters. In the event of any discrepancies, a third independent rater measured the sample(s), and the first authors
scores were retained based on above 85% agreement for all
measures (see the Reliability section).
The DEAP Inconsistency subtest was transcribed
using broad transcription following the manual (Dodd et al.,
2002). Responses to the Single-Word Test of Polysyllables
were transcribed from an audio recording into the PROPH
module in Computerized Profiling (Long, Fey, & Channell,
2006). Transcription included broad transcription supplemented with International Phonetic Alphabet diacritics to
mark distortions where clearly present. Measures extracted
from PROPH were percentage consonants correctrevised
(PCC-R; Shriberg, Austin, Lewis, McSweeny, & Wilson,
1997), percentage vowels correct (PVC), percentage phonemes
correct (PPC), occurrences of intrusive schwa, distorted
substitutions and voicing errors, and percentage of syllable
stress matches compared to the gloss for each word. Syllable
segregation (transcribed as a plus sign + between syllables) and intrusive schwas between cluster elements were
recorded by simple counts of occurrence.
PCC-R is a revised metric of PCC in which typical and
atypical distortions are removed from the index (Shriberg,
Austin, et al., 1997). PCC-R was used as a general index of
speech sound disorder severity, regardless of specific diagnosis, for all three groups. This metric was used in this study
because it was more sensitive for the participant sample with
a diverse range of age and speech status (Shriberg, Austin,
et al., 1997). Distortions were counted as their own measure
because it may facilitate differential diagnosis of speech
disorders. Thus, using PCC-R allowed us to identify any
potential effects related to general severity rather than a specific diagnosis of CAS. Accordingly, distortions were not
part of the PCC-R measure, yet were measured separately
under the Occurrence of Consonant and Vowel Distortions
Including Distorted Substitutions measurement according
to Shriberg, Potter, and Strand (2009).
The Oral and Motor Speech Protocol and connected
speech sample were transcribed or scored from a video recording. Articulation rate was calculated by counting syllables per second produced in 1 min of monologue connected
speech. This was undertaken using Adobe Audition software
(Version 1.5), in which the sample was isolated and vowels
identified and counted to determine syllable rate following
Logan, Byrd, Mazzocchi, and Gillam (2011).
Strands criterion of increased difficulty with polysyllabic words (Shriberg, Potter, et al., 2009) was determined
using a magnitude-of-change score. This score was generated for each participant, comparing the mean number of
Reliability
Intrarater and interrater transcription reliability was
calculated on a point-to-point basis for all unstandardized
dependent measures for each participant. Intrarater reliability was calculated on 20% of each measure for every participants data with each rater calculating the measure once
and then a second time at least 2 weeks after the initial calculation. Intrarater reliability for the first author was 94.6%
(SD = 3.6, range = 8898) and across all nine independent
raters was 95.2% (SD = 3.1, range = 88.399.1). Interrater
reliability was calculated first between the first author
and the nine raters and then between the nine independent
raters. Mean interrater reliability with the first author across
all measures was 94.2% (SD = 1.7, range = 91.396.2) and
between the nine raters was 93.3% (SD = 2.9, range = 86.4
98.1). For a breakdown of each raters and measures reliability, see online Supplemental Appendices 1 and 2.
Statistical Analyses
Simple bivariate followed by hierarchical multivariate, discriminant function analysis (DFA; McKean &
Hettmansperger, 1976) was used to determine whether one
or more of the 24 quantitative measures could reliably predict
the expert assignment of children to CAS or non-CAS
groups. Therefore, the results from the expert judgment of
CAS presence or absence and the quantitative measures were
compared in this phase to address the primary research aim.
The sample size was sufficient to minimize Type I
and Type II errors (Serlin & Harwell, 2004). Before completion of the analysis, data were screened for normality, linearity, and homoscedasticity. No violations of assumptions
were noted. Mahalanobis Distance was calculated to check
for outliers (Allen & Bennett, 2008) and did not exceed the
critical c2 of 26.13 (df = 8; a = .001) for any cases. Finally,
screening for multicollinearity above a conservative 0.80
(cf. 0.90; Allen & Bennett, 2008; Poulsen & French, 2004;
Tabachnick & Fidell, 2007) identified the following highly
correlated measures: PVC, PCC-R, and PPC; PVC and percentage stress matches; PPC and oral function score; PCC-R
and oral function score, percentage stress matches and
presence of articulatory false starts, restarts and/or withinspeech groping; and finally, /p/, /t/, and /k/ rates. When
two measures showed multicollinearity, only one measure
at a time was entered into the DFA as a predictor. There is
no consensus on the minimum ratio of predictor variables
to cases in DFA. Given this, we followed guidelines by
Poulsen and French (2004) and Tabachnick and Fidell
49
Results
50
Discussion
The purpose of the current study was to determine
whether expert diagnosis of CAS in a sample of 47 children
Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015
Table 3. Model 1: Results from bivariate DFA for all measures in order from highest to lowest accuracy in discriminating CAS/CAS+ group from
the non-CAS group participants.
Measure
Syllable segregation
Percentage of stress matches (lexical stress) on polysyllable test
Presence of false articulatory starts, restarts, or within-speech groping
DEAP (Inconsistency subtest)
PVC on the polysyllable test
/ptk/ accuracy
Nonspeech groping movements
PPC on the polysyllable test
Oral structure score
Intrusive schwa
/k/ rate
Articulation rate (syllables per second) in connected speech
Oral function score
PCC-R on the polysyllable test
/ptk/ rate
/t/ rate
/ptikek / rate
Vowel or consonant distortions
Maximum phonation time /a / (MPT)
CELF receptive language score
CELF expressive language score
/p/ rate
Voicing errors
Increased difficulty with polysyllable words (change score)
Bivariate DFA
Accuracy in diagnosing
CAS /CAS+ vs. non-CAS
0.890
0.882
0.764
0.551
0.546
0.535
0.536
0.515
0.492
0.477
0.445
0.428
0.386
0.389
0.356
0.352
0.321
0.067
0.261
0.270
0.142
0.291
0.231
0.205
<.001
<.001
<.001
<.001
<.001
<.001
.002
<.001
.004
<.001
.002
.006
.007
.007
.014
.024
.028
.669
.091
.080
.365
.058
.136
.196
79%
77%
58%
30%
30%
29%
29%
27%
24%
23%
17%
17%
15%
15%
13%
12%
10%
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
Note. DFA = discriminant function analysis; DEAP = Diagnostic Evaluation of Articulation and Phonology; PVC = percentage vowels correct;
PPC = percentage phonemes correct; PCC-R = percentage consonants correctrevised.
Table 4. Model 2: Results from bivariate DFA for all measures in order from highest to lowest accuracy in discriminating the CAS group
(excluding CAS+) from the non-CAS group (excluding submucous cleft) participants.
Measure
Percentage of stress matches (lexical stress) on polysyllable test
Syllable segregation
Presence of false articulatory starts, restarts, or within-speech groping
DEAP (Inconsistency subtest)
PVC on the polysyllable test
/ptk/ accuracy
Nonspeech groping movements
PPC on the polysyllable test
Oral function score
Vowel or consonant distortions
Intrusive schwa
PCC-R on the polysyllable test
Articulation rate (syllables per second) in connected speech
/k/ rate
CELF receptive language score
/t/ rate
Oral structure score
/ptk/ rate
/ptikek / rate
Maximum phonation time /a / (MPT)
CELF expressive language score
/p/ rate
Voicing errors
Increased difficulty with polysyllable words (change score)
Bivariate DFA
Accuracy in diagnosing
CAS vs. non-CAS
0.893
0.873
0.732
0.670
0.615
0.606
0.507
0.540
0.467
0.464
0.449
0.429
0.429
0.388
0.362
0.326
0.280
0.258
0.214
0.166
0.215
0.269
0.237
0.208
<.001
<.001
<.001
<.001
<.001
<.001
.002
<.001
.002
.003
.004
.007
.006
.013
.022
.040
.081
.109
.185
.306
.183
.093
.141
.199
80%
76%
54%
45%
38%
37%
26%
29%
22%
22%
20%
18%
18%
15%
13%
12%
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
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Table 5. Unstandardized (B) and standardized () regression coefficients for each predictive measure in a regression model predicting CAS
diagnosis.
Variable/measure
(Constant)
Percentage of stress matches on
polysyllable test
Syllable segregation
(Constant)
Percentage of stress matches on
polysyllable test
Syllable segregation
PPC on polysyllable test
Accuracy on DDK task /ptk/
0.707
0.009
0.575
Participants included
Model 1
CAS and CAS+ group (n = 32) vs.
non-CAS (n = 15)
Model 2
CAS (n = 28) vs. non-CAS (n = 12)
(CAS+ and submucous cleft
omitted)
0.382
p
<.001
<.001
0.012
0.504
0.011
0.729
.001
<.001
<.001
0.012
0.007
0.003
0.383
0.300
0.145
<.001
.001
.039
Accuracy in diagnosing
CAS in this sample
82%
91%
a
The values indicate the contribution (weight) of each task in diagnosis of CAS. A high score on a positive coefficient predicts CAS is more
likely, whereas, on a negative coefficient, a low score predicts CAS is more likely. Taking all the measures and weightings together gives the
overall accuracy in diagnosing CAS in this sample.
could be predicted from a combination of quantitative measures often collected as part of standard clinical practice.
The current standard for diagnosing CAS utilizes experts
making judgments of presence or absence of a small set
of speech behaviors, with no operational definitions or standardized testing protocol for eliciting the behaviors. In
the current sample, 28 children were judged by this method
to have CAS ( expressive language disorder; ASHA,
2007b; Shriberg et al., 2012), four were judged to have CAS+
(CAS plus dysarthria and receptive and/or expressive language
impairment), and 15 were judged to have either submucous
Participant characteristic
0.011
6
0.012
45
0.007
46
0.003
77
Raw score
33
20
25
66
Raw score
26
19
79
72
Raw score
88
97
100
Weighting ( values)
Raw score
Formula as entered
into Excel
Resulta
= round(0.504 + (6 0.011)
+ (45 0.012) + (46 0.007)
+ (77 0.003), 0)
= round(0.504 + (33 0.011)
+ (2 0.012) + (25 0.007)
+ (66 0.003), 0)
Note. Note the importance of the whole pattern of responses, rather than considering cutoffs variable by variable. Participants No. 2216 and
no. 2219, both diagnosed with CAS, had better scores for PPC on the polysyllable test than Participant No. 1311, diagnosed without CAS.
Classification of Participants No. 2216 and No. 2219 as CAS, using the formula, occurs because their substantially poorer scores on percentage
of stress matches on polysyllable test, syllable segregation, and accuracy on DDK task tip them over the cutoff for CAS classification.
Participant No. 1311s better scores on these three measures and his mildly compromised PPC on the polysyllable test all indicate he does not
fit the pattern of CAS in this sample.
a
52
Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015
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Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015
instances of syllable segregation, and poorer /ptk/ accuracy than the boy with phonological impairment. These
three features are hypothesized to reflect an underlying impairment of motor planning and programming or praxis
(e.g., ASHA, 2007b; Shriberg, Green, et al., 2003; Thoonen
et al., 1999). The criteria provide confidence in discriminating those with some underlying praxis deficits, demonstrating CAS is at least part of the childs presentation.
Therefore, the combination of the four discriminant measures
and their weightings appear crucial in separating verbal
participants with different types of expressive communication disorder and wide ranges of age and severity.
It is important to consider that the assessment procedures used here assessed children at a single time point.
Any developmental disorder can vary in its surface presentation across time. Regular review is therefore indicated to
ensure early detection of other clinical behaviors that might
emerge with therapy and maturation (e.g., literacy concerns)
and ultimately to ensure that management strategies match
presenting and prognostic features (Stackhouse & Snowling,
1992; Zaretsky, Velleman, & Curro, 2010).
Our findings advance the field in this area by identifying two tests that appear central to differentiating CAS
from other disorders, at least in this sample: namely, a complete OMA including a DDK task and a sufficiently large
sample of polysyllabic single-word production (e.g., Gozzard
et al., 2004). Both the real-word polysyllabic test and the
nonword /ptk/ DDK task from the OMA were motorically challenging and appear to successfully elicit behaviors
that reflected the underlying motor planning and programming deficits in CAS at both the segmental and suprasegmental level (Shriberg, Lohmeier, et al., 2009; Thoonen
et al., 1996, 1999). These two tests alone may be sufficient
for reliable diagnosis of CAS in verbal children. However,
SLPs using these tasks still need to consider normal acquisition and development in terms of both segmental and prosody
accuracy (Ballard, Djaja, Arciuli, James, & van Doorn,
2012; James, 2006; Shriberg, Lohmeier, et al., 2009).
Limitations
The major limitation of this study is that it utilized
a selected clinical sample of children with suspected CAS
and selection criteria designed to find idiopathic CAS
rather than comorbid CAS. This approach is not uncommon (e.g., McCauley et al., 2012; Rosenbek & Wertz, 1972;
Yoss & Darley, 1974) and was warranted in exploring and
identifying quantitative measures for further investigation.
As in any multivariate DFA, the results are heavily reliant
on the characteristics of the sample used and the measures
selected for analysis, and there are inherent risks that the
findings might not generalize to a similar but larger sample
or to a different group of children (e.g., children with comorbid intellectual disability and receptive language impairment). For example, the formula used PPC to separate
CAS from ataxic dysarthria. However, there is no reason
that a child with CAS may not have severe articulation
errors. Also, the participant sample here all demonstrated
prosodic errors being features on both the ASHA consensusbased (2007b) and Strand 10-point feature lists (Shriberg,
Potter, & Strand, 2009). However, other samples that demonstrate dysprosody may only account for a subset of children with CAS (e.g., Shriberg, Aram, & Kwiatkowski, 1997)
or younger children may demonstrate other errors of greater
prevalence, such as inconsistency (Iuzzini & Forrest, 2010).
The Model 2 DFA formula based on the limited data set is
reported here to facilitate replication, but caution must be
used in applying it to clinical populations or cases that differ
from the studied sample.
In this study, we relied on perceptual measures of
speech behaviors. This was a deliberate decision considering
that perceptual measures are most commonly used in clinical practice (Duffy, 2012) and have been shown to correlate
highly with acoustic measures of lexical stress (e.g., pairwise variability indices; Ballard et al., 2010). However, the
relatively subjective nature of these measures could result
in discrepancies and therefore errors in the formulas result,
particularly in untrained SLPs. In the current study, training of raters was required to maintain interrater reliability
above 85%. To assist replication in future studies, efforts to
make the measures more objective would be warranted by
providing defined features, measures, and scoring criteria in
a manual or by finding more objective kinematic or acoustic measures. There are promising acoustic measures (e.g.,
Ballard et al., 2012; McKechnie et al., 2008; Shriberg, Green,
et al., 2003) and potential for automation (Hosom, Shriberg,
& Green, 2004; Rosen et al., 2010). However, manual
acoustic measures can be time-intensive and also require a period of training. It may be some time before such acoustic
measures are available in a format that allows rapid automated analysis in clinical settings.
Studies aiming to identify quantitative measures that
differentiate disorders suffer an inherent circularity (ASHA,
2007b; Guyette & Diedrich, 1981). However, here the aim
was not to validate the original checklists (ASHA, 2007b;
Shriberg et al., 2009) used for initial diagnosis. Instead, the
aim was to test the assessment samples to determine whether
a more replicable and efficient method (or set of measures
in combination) could reproduce the initial expert diagnosis
with high accuracy. Our findings suggest that a set of just
four measures strongly predicts presence of CAS in verbal
children. This is a more parsimonious solution than a
checklist of multiple features, of which only a subset is required for diagnosis (e.g., Hickman, 1997; Kaufman, 1995;
Shriberg, Potter, & Strand, 2009).
Further Directions
This preliminary study used a sample of children ages
412 years with idiopathic and comorbid CAS and compared them to others who had been suspected to have CAS
but were instead diagnosed with phonological or language
impairment, submucous cleft, or dysarthria. Further research is necessary to test the robustness of the sets of criteria and the resultant formula identified when diagnosing
an unselected community sample of children with suspected
55
Conclusions
This study commenced with expert diagnosis of children with suspected CAS and then extracted quantitative
measures that were analyzed to determine whether any combination predicted the expert diagnosis. CAS and non-CAS
in verbal 4- to 12-year-olds in this sample could be discriminated with 91% accuracy based on four measures, following completion of a thorough OMA including accuracy on
/ptk/ (Robbins & Klee, 1987) and a 50-word sample
of polysyllable words (Gozzard et al., 2004). The results met
Shribergs criteria of >90% sensitivity and specificity, but
within a selected sample. A formula based on the results of
DFA is provided to assist in application and replication.
Further research is required with a larger unselected sample to ensure that the four measures and the resultant formula can differentiate CAS in a wider population from
typical development and other speech, neurological, and
linguistic disorders. Investigation of kinematic, acoustic,
and other assessment tools for CAS features would also
be beneficial.
Acknowledgments
This research was supported by the Douglas and Lola Douglas
Scholarship on Child and Adolescent Health, the Nadia Verrall
Memorial Research Grant (2010), a Postgraduate Research Award
(2011) from Speech Pathology Australia, and the James Kentley
Memorial Scholarship and Postgraduate Research Support Schemes
to Elizabeth Murray; a University of Sydney International Program Development Fund Grant to Patricia McCabe and Kirrie
Ballard; and an Australian Research Council Future Fellowship
(FT120100255) to Kirrie Ballard. Parts of this article were presented
at the Motor Speech Conference in Santa Rosa, CA, 2012, and the
Speech Pathology Australia Conference in Hobart, Tasmania, 2012.
We thank research assistants Morin Beausoleil, Virginia Caravez,
Claire Formby, Jennifer Fortin Zornow, Sally Hanna, Claire Layfield,
Aimee-Kate Parkes, Gemma Patterson, Alyssa Piper, and Caitlin
Winkelman. Thank you also to Samantha Warhurst, Kate Anderson,
and Claire Layfield for editorial suggestions.
56
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Appendix A
Diagnostic results for all participants using the ASHA criteria (2007b) and the Strand 10-point checklist (Shriberg, Potter, &
Strand, 2009).
Participant no.
ASHA
criteria meta
Strand
criteria metb
1217
1314
2/3
2/3
5/10
6/10
0236
1313
0235
0/3
1/3
0/3
1/10
1/10
0/10
0333
0/3
3/10
0/3
0/3
0/3
0/3
0/3
1/3
1/3
1/3
0/3
0/15
1/10
2/10
2/10
1/10
2/10
6/10
2/10
2/10
1/10
3/15
3/3
7/10
2105
1214
1328
3/3
3/3
3/3
8/10
9/10
8/10
3/3
8/10
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
28/28
8/10
7/10
8/10
6/10
9/10
7/10
8/10
8/10
9/10
8/10
7/10
6/10
7/10
7/10
7/10
5/10
7/10
5/10
8/10
5/10
5/10
7/10
6/10
6/10
7/10
7/10
5/10
6/10
28/28
Group
Non-CAS
0334
0237
1311
2315
1312
0138
0139
1310
0335
No. of non-CAS participants
that met criteria
CAS+
0332
Diagnosis
Ataxic dysarthria (+ receptive & expressive language disorder)
Flaccid dysarthria (CNXII) (+ receptive & expressive language
disorder)
Phonological disorder (+ expressive language disorder)
Phonological disorder
Phonological disorder (+ receptive & expressive language disorder);
hoarse voice
Phonological disorder (+ receptive & expressive language disorder);
suspected global developmental delay
Phonological disorder (+ receptive & expressive language disorder)
Phonological disorder (+ receptive & expressive language disorder)
Phonological disorder
Phonological disorder
Comorbid phonological disorder and stuttering
Submucous cleft palate (+ expressive language disorder)
Submucous cleft palate
Submucous cleft palate
Phonological disorder (+ expressive language disorder)
CAS and ataxic dysarthria (+ receptive & expressive language
disorder)
CAS and flaccid dysarthria (CNXII) (+ expressive language disorder)
CAS and flaccid dysarthria (CNXII) (+ expressive language disorder)
CAS, flaccid dysarthria (CNXII) (+ receptive & expressive language
disorder)
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
ASHA criteria = 3/3 needed for CAS diagnosis. bStrand criteria = 4/10 needed for CAS diagnosis over three tasks.
59
Appendix B
Sensitivity and specificity based on the Model 2 DFA.
Model 2 participants
Expert diagnosis
CAS (n = 28) (excluding CAS+)
True positive = 100% (n = 28)
False negative = 0% (n = 0)
All participants
Expert diagnosis
CAS (n = 32) (including CAS+)
True positive = 97% (n = 31)
False negative = 3% (n = 1)
60
Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015
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